Miltefosine-related paracentral ulcerative keratolysis in a patient with active cutaneous leishmaniasis from Nepal

Dear Sir,

Miltefosine (MF) is widely used in several countries—including India, Bangladesh and Nepal—under the kala-azar elimination programme. ¹ With its use on the rise, so are the reports of related ocular complications. These have already been reported from India and Bangladesh; we would like to contribute to this record from Nepal, where we encountered a similar case.

A 15-year-old girl presented with diffuse redness, pain and photophobia in both eyes. Written consent was obtained from the patient and her parent for reporting and publishing this case. Patient confidentiality is maintained. The girl is a permanent resident of Siraha, in the eastern flatland of Nepal. She was under treatment with MF for four weeks as an outpatient from a regional hospital before she started developing ocular symptoms. Her vision was light perception in both her eyes and more could not be done due to blepharospasm. On examination, she had ring-shaped peripheral ulcerative keratolysis with infiltration bilaterally involving the sclera and had developed corneal infiltration in both eyes (Figure 1a and 1b). She also had perioral dermal lesions (Figure 1c). She had started topical moxifloxacin after visiting a local eye centre before referral to our hospital. We suspected a drug-induced keratopathy; therefore, we advised stopping MF and started her on liposomal amphotericin B (AmBisome) 2.5 mg/kg intravenously for 20 days after consulting an expert in infectious diseases. We advised continuing moxifloxacin but added prednisolone acetate q.d.s., doxycycline 100 mg b.i.d., vitamin C 500 mg t.d.s. OPEN IN VIEWER

On follow-up, she was symptomatically better after one week, with vision of 3/60 in the right and 2/60 in the left eye. We advised her to continue the same medication. Follow-up at two weeks revealed a healing corneal lesion with subsiding scleral inflammation (Figure 2). We advised completion of the AmBisome dose with the same topical medication as prescribed for a further two weeks. OPEN IN VIEWER

However, she presented with worsening of her condition after seven days. Her vision had deteriorated to hand movement bilaterally. The corneal infiltrates and inflammation had increased remarkably (Figure 3). She told us that MF had been restarted due to concerns of incomplete treatment by her local medical centre; consequently, we advised a complete halt to the MF treatment and continuation of AmBisome; we also increased the frequency of prednisolone acetate to every 2 h. We left the rest of the medication as it was. OPEN IN VIEWER

Follow-up on days 35, 45 and 60 from the initial presentation (Figure 4) showed her ocular signs had significantly improved and there was resolution of keratolysis with paralimbal scarring. Her vision improved from hand movement to 6/12 over this period. OPEN IN VIEWER

It has been already been indicated that amphotericin B can be used for the treatment of post-kala-azar dermal leishmaniasis^2,3 and this holds true for this case. The fact that the symptoms improved after stopping MF and flared up again after initiation of the drug is a predictable sign that these corneal lesions are related to MF.

It is very important to recognise that in our part of the world a corneal ulcer or scleritis-like lesion in patients who are under treatment for kala-azar or its complications could be drug-related. Stopping the offending agent and switching to alternative medication can be safely done in such cases with good outcomes if identified early. An addition of topical steroids and systemic doxycycline may be of value.